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达沙替尼相关乳糜胸在一名慢性粒细胞白血病儿科患者中的病例报告及文献综述

Dasatinib-associated chylothorax in a pediatric patient with chronic myeloid leukemia: a case report and literature review.

作者信息

Liu Qinqin, Fu Jinqiu, Zhang Aijun

机构信息

Department of Pediatrics, Qilu Hospital of Shandong University, Jinan, China.

出版信息

Transl Cancer Res. 2023 Jan 30;12(1):194-200. doi: 10.21037/tcr-22-1983. Epub 2023 Jan 6.

Abstract

BACKGROUND

Dasatinib is an effective 2 generation tyrosine kinase inhibitor for the treatment of newly diagnosed or intolerant to imatinib chronic myeloid leukemia (CML), and in Philadelphia chromosome (Ph)-positive acute lymphoblastic leukemia (ALL). The most common adverse effects of dasatinib include gastrointestinal upset, pancytopenia, skin rash, diarrhea and fluid retention. Pleural effusion (PE), which occurs in almost 15-35% of patients, is the most frequent manifestation of fluid retention. However, Dasatinib-induced chylothorax is extremely rare. There are solely 13 cases of dasatinib-related chylothorax in adults in the literature, while only one pediatric patient has been reported. The preferred treatment options are usually with systemic steroids, diuretics, and dasatinib discontinuation. We report the second pediatric case and propose the hypothesis of its mechanism and summarize the relevant cases to facilitate the understanding of the pathophysiology, clinical manifestation, management and prognosis of dasatinib-induced chylothorax.

CASE DESCRIPTION

An 11-year-old boy diagnosed with breakpoint cluster region-Abelson (BCR-ABL) fusion was treated with dasatinib. After 38 months, the patient was admitted for dyspnea characterized by decreased breath sounds on both lungs during physical examination. Computed tomography (CT) showed bilateral PE with local insufficiency of both lungs. Drug-induced chylothorax was presumed based on clinical manifestations, excluding other possible causes. Dasatinib was withdrawn, diuretics as well as steroids were given for supportive therapy and octreotide was administered to decrease fat absorption in the intestine. However, the chylous fluid did not decrease significantly. The patient was then being fasted. Unexpectedly, after fasted for two days, the chylous fluid became clear and the drainage volume was decreased. The patient was advised to use nilotinib. We followed up the patient for 8 months, and there was no recurrence of chylothorax.

CONCLUSIONS

Our patient had a shorter treatment course for chylothorax than those in the literature. In addition to dasatinib withdrawal, fasting treatment was also utmost critical. We summarize the literature of known existing cases to improve the understanding of the side effects and management of dasatinib in the treatment of CML.

摘要

背景

达沙替尼是一种有效的二代酪氨酸激酶抑制剂,用于治疗新诊断的或对伊马替尼不耐受的慢性髓性白血病(CML),以及费城染色体(Ph)阳性的急性淋巴细胞白血病(ALL)。达沙替尼最常见的不良反应包括胃肠道不适、全血细胞减少、皮疹、腹泻和液体潴留。胸腔积液(PE)在近15% - 35%的患者中出现,是液体潴留最常见的表现。然而,达沙替尼引起的乳糜胸极为罕见。文献中仅有13例成人达沙替尼相关乳糜胸病例,而小儿患者仅报道过1例。首选的治疗方案通常是全身使用类固醇、利尿剂,并停用达沙替尼。我们报告第二例小儿病例,并提出其机制假说,总结相关病例,以促进对达沙替尼引起乳糜胸的病理生理学、临床表现、管理和预后的理解。

病例描述

一名11岁诊断为断裂簇集区 - 阿贝尔森(BCR - ABL)融合的男孩接受达沙替尼治疗。38个月后,患者因呼吸困难入院,体格检查发现双肺呼吸音减弱。计算机断层扫描(CT)显示双侧胸腔积液伴双肺局部肺不张。根据临床表现推测为药物性乳糜胸,排除了其他可能原因。停用达沙替尼,给予利尿剂和类固醇进行支持治疗,并使用奥曲肽减少肠道脂肪吸收。然而,乳糜液并未显著减少。随后患者禁食。出乎意料的是,禁食两天后,乳糜液变清,引流量减少。建议患者使用尼罗替尼。我们对患者随访了8个月,乳糜胸未复发。

结论

我们的患者乳糜胸的治疗疗程比文献报道的患者短。除停用达沙替尼外,禁食治疗也至关重要。我们总结了已知现有病例的文献,以提高对达沙替尼治疗CML时副作用及管理的认识。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8b08/9906050/db68d4db1229/tcr-12-01-194-f1.jpg

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